{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,30]],"date-time":"2026-07-30T18:43:25Z","timestamp":1785437005837,"version":"3.56.0"},"reference-count":12,"publisher":"American Society for Microbiology","issue":"4","license":[{"start":{"date-parts":[[2025,7,1]],"date-time":"2025-07-01T00:00:00Z","timestamp":1751328000000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2025,7,1]],"date-time":"2025-07-01T00:00:00Z","timestamp":1751328000000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/journals.asm.org\/non-commercial-tdm-license"}],"funder":[{"name":"EU funding Next generation EU MUR","award":["PE00000007"],"award-info":[{"award-number":["PE00000007"]}]}],"content-domain":{"domain":["journals.asm.org"],"crossmark-restriction":true},"short-container-title":["ASM Case Rep"],"published-print":{"date-parts":[[2025,7]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:sec>\n                    <jats:title\/>\n                    <jats:sec>\n                      <jats:title>Background<\/jats:title>\n                      <jats:p>\n                        Individuals pharmacologically immunosuppressed, whether by chemical or biological drugs, are prone to severe infections, including leishmaniasis. In\n                        <jats:italic toggle=\"yes\">Leishmania<\/jats:italic>\n                        -infected immunocompromised patients, numerous factors contribute to the clinical picture and response to treatment, including the parasite, the immune system, and the co-medications.\n                      <\/jats:p>\n                    <\/jats:sec>\n                    <jats:sec>\n                      <jats:title>Case Summary<\/jats:title>\n                      <jats:p>\n                        Here, we present the case of a 2-year relapsing visceral leishmaniasis (VL) in an immunocompromised patient suffering from undifferentiated connective tissue disease and undergoing treatment for osteoporosis with a monoclonal antibody targeting the bone resorption mediator receptor activator of nuclear factor-\u03baB ligand (RANKL). The ultimate resolution of VL was achieved by an integrated approach that included the administration of combined antiparasitic treatment followed by secondary prophylaxis, tapering of immunosuppression including drastic reduction of oral steroids and suspension of hydroxychloroquine and the suspension of RANKL inhibitor. As RANKL restrains\n                        <jats:italic toggle=\"yes\">Leishmania<\/jats:italic>\n                        replication by activating macrophages, we hypothesize that the RANKL inhibitor may have synergized with the immunosuppressive treatment in inducing parasite expansion and relapsing of VL in the index case.\n                      <\/jats:p>\n                    <\/jats:sec>\n                    <jats:sec>\n                      <jats:title>Conclusion<\/jats:title>\n                      <jats:p>\n                        This case highlights the need to consider\n                        <jats:italic toggle=\"yes\">Leishmania<\/jats:italic>\n                        -infected immunocompromised patients as complex systems; numerous factors can be implicated in the balance between the parasite, the immune system, and co-medications and may contribute to the response to treatment.\n                      <\/jats:p>\n                    <\/jats:sec>\n                  <\/jats:sec>","DOI":"10.1128\/asmcr.00006-25","type":"journal-article","created":{"date-parts":[[2025,5,8]],"date-time":"2025-05-08T09:00:14Z","timestamp":1746694814000},"update-policy":"https:\/\/doi.org\/10.1128\/asmj-crossmark-policy-page","source":"Crossref","is-referenced-by-count":0,"title":["A case of relapsing visceral leishmaniasis: the potential role of steroids and RANKL inhibitors in worsening disease outcome"],"prefix":"10.1128","volume":"1","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-0687-9493","authenticated-orcid":false,"given":"Irene","family":"Zaghi","sequence":"first","affiliation":[{"id":[{"id":"https:\/\/ror.org\/01111rn36","id-type":"ROR","asserted-by":"publisher"}],"name":"Department of Medical and Surgical Sciences, University of Bologna","place":["Bologna, Italy"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Margherita","family":"Ortalli","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/01111rn36","id-type":"ROR","asserted-by":"publisher"}],"name":"Department of Medical and Surgical Sciences, University of Bologna","place":["Bologna, Italy"]},{"name":"Unit of Microbiology, IRCCS Azienda Ospedaliero-Universitaria di Bologna","place":["Bologna, Italy"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alessandra","family":"Mistral De Pascali","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/01111rn36","id-type":"ROR","asserted-by":"publisher"}],"name":"Department of Medical and Surgical Sciences, University of Bologna","place":["Bologna, Italy"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bianca","family":"Granozzi","sequence":"additional","affiliation":[{"name":"Infectious Diseases Unit, IRCCS Azienda Ospedaliero-Universitaria di Bologna","place":["Bologna, Italy"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alessandra","family":"Cascavilla","sequence":"additional","affiliation":[{"name":"Infectious Diseases Unit, IRCCS Azienda Ospedaliero-Universitaria di Bologna","place":["Bologna, Italy"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Luciano","family":"Attard","sequence":"additional","affiliation":[{"name":"Infectious Diseases Unit, IRCCS Azienda Ospedaliero-Universitaria di Bologna","place":["Bologna, Italy"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alessandro","family":"Deni","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/01111rn36","id-type":"ROR","asserted-by":"publisher"}],"name":"Department of Medical and Surgical Sciences, University of Bologna","place":["Bologna, Italy"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Stefania","family":"Varani","sequence":"additional","affiliation":[{"id":[{"id":"https:\/\/ror.org\/01111rn36","id-type":"ROR","asserted-by":"publisher"}],"name":"Department of Medical and Surgical Sciences, University of Bologna","place":["Bologna, Italy"]}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"235","reference":[{"key":"e_1_3_3_2_2","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pntd.0011497"},{"key":"e_1_3_3_3_2","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pntd.0006375"},{"key":"e_1_3_3_4_2","doi-asserted-by":"publisher","DOI":"10.1007\/s12325-021-01936-y"},{"key":"e_1_3_3_5_2","doi-asserted-by":"publisher","DOI":"10.2807\/1560-7917.ES.2024.29.4.2300190"},{"key":"e_1_3_3_6_2","doi-asserted-by":"publisher","DOI":"10.1007\/s15010-022-01943-3"},{"key":"e_1_3_3_7_2","doi-asserted-by":"publisher","DOI":"10.1590\/s0100-879x2004000400020"},{"key":"e_1_3_3_8_2","unstructured":"Luigi G Rogelio L-V Mourad M. 2017. Manual on case management and surveillance of the leishmaniases in the WHO European region. Manual on case management and surveillance of the leishmaniases in the WHO European region."},{"key":"e_1_3_3_9_2","doi-asserted-by":"publisher","DOI":"10.1016\/S1473-3099(23)00353-5"},{"key":"e_1_3_3_10_2","doi-asserted-by":"publisher","DOI":"10.3389\/fendo.2023.1182753"},{"key":"e_1_3_3_11_2","doi-asserted-by":"publisher","DOI":"10.1210\/clinem\/dgz322"},{"key":"e_1_3_3_12_2","doi-asserted-by":"publisher","DOI":"10.1111\/1756-185X.14101"},{"key":"e_1_3_3_13_2","doi-asserted-by":"publisher","DOI":"10.3389\/fimmu.2021.704429"}],"container-title":["ASM Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/journals.asm.org\/doi\/pdf\/10.1128\/asmcr.00006-25","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.asm.org\/doi\/pdf\/10.1128\/asmcr.00006-25","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,9,9]],"date-time":"2025-09-09T17:29:14Z","timestamp":1757438954000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.asm.org\/doi\/10.1128\/asmcr.00006-25"}},"subtitle":[],"editor":[{"given":"Ritu","family":"Banerjee","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"editor"}]}],"short-title":[],"issued":{"date-parts":[[2025,7]]},"references-count":12,"journal-issue":{"issue":"4","published-print":{"date-parts":[[2025,7]]}},"alternative-id":["10.1128\/asmcr.00006-25"],"URL":"https:\/\/doi.org\/10.1128\/asmcr.00006-25","relation":{},"ISSN":["2996-2684"],"issn-type":[{"value":"2996-2684","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,7]]},"assertion":[{"value":"2025-01-07","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2025-04-02","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2025-05-08","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}],"article-number":"e00006-25"}}